| Summary: | Key Clinical Message This case report highlights the rarity of distal tracheal adenoid cystic carcinoma and emphasizes the importance of considering it as a differential diagnosis in patients presenting with sustained dyspnea. Early diagnosis and a multidisciplinary approach involving thoracic surgeons, radiation oncologists, and medical oncologists are crucial for optimal treatment planning and patient outcomes. Further research is warranted to better understand the pathogenesis, molecular characteristics, and optimal management strategies for this rare tracheal malignancy. Abstract Adenoid cystic carcinoma (ACC) is an unusual malignant neoplasm that mostly arises in the minor salivary glands. It can also occur in various mucous membrane‐lined structures, including the trachea. The treatment of choice is surgery in resectable cases, but radiotherapy and chemotherapy are generally utilized for unresectable cases or palliative treatment. In this study, I report my experience with a case of unresectable ACC occurring in a distal trachea treated with concurrent chemoradiotherapy (CCRT), based on the patient's clinical presentation, diagnostic workup, histopathological findings, treatment modalities, and long‐term prognosis.
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